A 25-year-old immunocompetent Japanese man was referred to us for investigation and treatment of obstructive jaundice. Computed tomography (CT) showed dilatation of the bilateral intrahepatic bile ducts and a low-attenuated mass extending along the portal tract, from the hepatic hilum to the periphery, suggestive of cholangiocellular carcinoma (CCC) with periductal infiltration. However, the culture of bile obtained by percutaneous transhepatic biliary drainage revealed Cryptococcus neoformans. Microscopic examination of a biopsy specimen of the mass-like lesion in the liver revealed fungal organisms encapsulated in multinucleated giant cells. A small area of infiltration in the left lung was also detected on chest CT, and a diagnosis of liver and lung cryptococcosis was made. After 8 months of antifungal therapy, both the liver and lung lesions had vanished and the biliary stricture resolved. Unnecessary surgical intervention was avoided by an early and accurate diagnosis and effective antifungal therapy. The bile culture was the key to the definitive diagnosis in this case.
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